BACKGROUND: Peliosis is a rare condition characterised by multiple cyst-like, blood-filled cavities within the parenchyma of solid organs, most commonly affecting the liver. Isolated splenic peliosis is an even more unusual phenomenon. Patients with AIDS may develop peliosis in association with bacillary angiomatosis. This is due to secondary infection with Bartonella henselae or a similar organism, Rochalimaea henselae. CASE PRESENTATION: A 45-year-old HIV-positive man on antiretroviral therapy presented with a left hypochodrial abdominal mass. Radiological and histopathological examination confirmed splenic peliosis.